Journal of Neuroscience · 2005 · 190 citations · 26 references
Life SciencesNeurobiologyNeuroscienceDevelopmental NeuroscienceMolecular NeurobiologyMolecular NeuroscienceGenetic EpilepsiesGlutamatergic SignalingMolecular NeurogeneticsNeurotransmissionSynaptic Strength RegulationSynaptic TransmissionEpileptiform ActivityEpileptiform DischargeExtracellular Signal-regulated Kinase PathwayExtracellular Signal-regulated Kinase SignalingFragile X Syndrome ModelsGlutamatergic SynapsesGlutamatergic Transmission ModulationMetabotropic Glutamate Receptor Signaling
Mutations in FMR1, which encodes the fragile X mental retardation protein (FMRP), are the cause of fragile X syndrome (FXS), an X-linked mental retardation disorder. Inactivation of the mouse gene Fmr1 confers a number of FXS-like phenotypes including an enhanced susceptibility to epileptogenesis during development. We find that in a FXS mouse model, in which the function of FMRP is suppressed, synaptically released glutamate induced prolonged epileptiform discharges resulting from enhanced group I metabotropic glutamate receptor (mGluR)-mediated responses in hippocampal slices. The induction of the group I mGluR-mediated, prolonged epileptiform discharges was inhibited in preparations that were pretreated with inhibitors of ERK1/2 (extracellular signal-regulated kinase 1/2) phosphorylation or of mRNA translation, and their maintenance was suppressed by group I mGluR antagonists. The results suggest that FMRP plays a key role in the control of signaling at the recurrent glutamatergic synapses in the hippocampus. The absence of this control causes the synaptically activated group I mGluRs to elicit translation-dependent epileptogenic activities.
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Fragile X-related protein and VIG associate with the RNA interference machinery
Amy A. Caudy, Mike M. Myers, Gregory J. Hannon et al. · Genes & Development · 2002 · 637 citations · Full text